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The expression of the two ERO1-L isoforms therefore appears to be differently regulated, in the way that ERO1-La expression is mainly controlled by the cellular oxygen tension, whilst ERO

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The cellular oxygen tension regulates expression of the endoplasmic oxidoreductase ERO1-La

Bernhard Gess1, Karl-Heinz Hofbauer1, Roland H Wenger2, Christiane Lohaus3, Helmut E Meyer3

and Armin Kurtz1

1

Institut fu¨r Physiologie der Universita¨t Regensburg, Germany;2Carl-Ludwig-Institut fu¨r Physiologie der Universita¨t Leipzig, Germany;3Medizinisches Proteom-Center der Ruhr-Universita¨t, Bochum, Germany

The formation of disulfide bonds in the endoplasmic

reti-culum requires protein disulfide isomerase (PDI) and

endoplasmic reticulum oxidoreductin 1 (ERO1) that

reoxi-dizes PDI We report here that the expression of the rat,

mouse and human homologues of ERO1-Like protein a but

not of the isoform ERO1-Lb are stimulated by hypoxia in

rats vivo and in rat, mouse and human cell cultures The

temporal pattern of hypoxic ERO1-La induction is very

similar to that of genes triggered by the hypoxia inducible

transcription factor (HIF-1) and is characteristically

mim-icked by cobalt and by deferoxamine, but is absent in cells

with a defective aryl hydrocarbon receptor translocator

(ARNT, HIF-1b) We speculate from these findings that

the expression of ERO1-La is probably regulated via the

HIF-pathway and thus belongs to the family of classic oxygen regulated genes Activation of the unfolded protein response (UPR) by tunicamycin, on the other hand, strongly induced ERO1-Lb and more moderately ERO1-La expres-sion The expression of the two ERO1-L isoforms therefore appears to be differently regulated, in the way that ERO1-La expression is mainly controlled by the cellular oxygen tension, whilst ERO1-Lb is triggered mainly by UPR The physiological meaning of the oxygen regulation of ERO1-La expression likely is to maintain the transfer rate of oxidizing equivalents to PDI in situations of an altered cellular redox state induced by changes of the cellular oxygen tension Keywords: hypoxia; HIF; protein folding; UPR; PDI

Formation of disulfide bonds is an essential event for the

correct folding of proteins in the endoplasmic reticulum

It is well known that this process is catalyzed by protein

disulfide-isomerase (PDI) [1] Until a few a years ago,

however, it remained unclear how PDI is reoxidized in this

reaction [2] It was the discovery of the ERO1

(endoplas-mic reticulum oxidoreductin) protein in yeast [3,4] which

provided evidence that this protein is essential to transfer

oxidizing equivalents to PDI [5] It turned out that ERO1

is a highly conserved endoplasmic protein and for humans

and mouse two ERO1-Like proteins have meanwhile been

identified, termed ERO1-La [6] and -1b [7] The ERO1

proteins are probably flavoproteins [8] that covalently bind

to PDI [9], what explains their function to transfer

oxidizing equivalents to PDI ERO1-La and -Lb display

different tissue distributions [7], and moreover, appear to

be differently regulated in their expression Thus, mainly

ERO1-L b transcripts are induced in the course of

unfolded protein response [7] In this pathway

accumula-tion of misfolded proteins in the endoplasmic reticulum

induces the expression of a number of proteins including those involved in the correct folding of proteins such as chaperones [10] How the expression of the ERO1-La protein is regulated is yet unknown Analyzing the protein expression pattern of a rat vascular smooth muscle cell line,

we now found that a ERO1-Like protein highly homo-logous to mouse and human ERO1-La is strongly upregulated during cellular hypoxia This study therefore aimed to characterize the effects of low oxygen tension on ERO1-L(a) expression

Materials and methods Cell cultures

Rat aortic vascular smooth muscle cells (A7r5) from BDXI rats (ATCC CRL 1444) were cultured in 75 cm2 flasks (Sarstedt) with 15 mL Dulbecco’s minimal essential medium (MEM) containing 10% fetal bovine serum and penicillin/streptomycin (10 U/10 lgÆmL)1, Biochrom), kept

in room air with 10% CO2at 37C Medium was changed every second day and cells were confluent on day 7–10 after splitting which was achieved with trypsin-EDTA for 5 min

at 37C For the experiments, cell cultures (triplicates) were incubated at room air (21% O2i.e normoxia) or 1%

O2 or 0.5% O2 (i.e hypoxia) for up to 12 h Additional culture dishes were incubated at 21% O2 with either cobaltous chloride (100 lmolÆL)1) or with deferoxamine (100 lmolÆL)1) for 12 h

To induce the unfolded protein response, A7r5 cells were incubated with 5 lgÆmL)1tunicamycin for 4.5, 8, 12 and

24 h

Correspondence to A Kurtz, Institut fu¨r Physiologie,

Universita¨t Regensburg, D-93040 Regensburg, Germany.

Fax: + 49 941 943 4315, Tel.: + 49 941 943 2980,

E-mail: armin.kurtz@vkl.uni-regensburg.de

Abbreviations: PDI, protein disulfide isomerase; ERO, endoplasmic

reticulum oxidoreductin; ERO1-L, ERO1-Like protein; HIF, hypoxia

inducible transcription factor; ARNT, aryl hydrocarbon receptor

translocator; UPR, unfolded protein response.

(Received 14 February 2003, revised 18 March 2003,

accepted 25 March 2003)

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Mouse hepatoma Hepa1 cells, and their subclone

Hepa1C4, which produces defective aryl hydrocarbon

receptor (ARNT, HIF-1b) [11] due to a point mutation

[12] rendering the cells unable to form active hypoxia

inducible factor HIF [13], were grown under the above

mentioned conditions For the experiments the cells were

incubated either at 0.5% O2(i.e hypoxia) or at 21% O2with

deferoxamine (100 lmolÆL)1) for 4.5 and 12 h

Human hepatoma HepG2 cells (used at 50% confluency)

and the mouse renin secreting cell line As4.1 [14] were also

grown under the above mentioned conditions The cells

were incubated either at 0.5% O2(i.e hypoxia) or at 21%

O2with deferoxamine (100 lmolÆL)1) for 4.5 h

In vivo experiments

All experiments were conducted in accordance with the

National Institutes of Health Guide for the Use of

Laboratory Animals and the German Law on the

protec-tion of Animals Male Sprague–Dawley rats (200–250 g)

that had free access to food and water were used for the

experiments and treated in the following way (a): in the

control group, animals received no treatment (n¼ 6) (b); in

the hypoxia group, the animals were placed in a gas-tight

box that was supplied continuously with a gas mixture of

8% O2-92%N2for 6 h (n¼ 6) (c); in the carbon monoxide

group, the animals were placed in a gas-tight box that

was supplied continuously with room-air plus 0.1% CO for

6 h (n¼ 6); and [4] for cobalt treatment, the rats were

subcutaneously injected with cobalt chloride (60 mgÆkg)1),

and the animals were killed 6 h later (n¼ 6) At the end of

the experiments, the animals were killed by decapitation

Aortas, brains, hearts, kidneys, livers and lungs were

removed quickly, weighed, and rapidly frozen in liquid

nitrogen All organs were stored at)80 C until isolation of

protein and total RNA

Preparation of protein samples

After removal of cell culture medium, cells were washed

three times with ice-cold NaCl/Piand then scraped off in

lysis buffer (300 lL per 75 cm2 flask) consisting of

7 molÆL)1urea, 2 molÆL)1thiourea, 2% Chaps, 1%

dithio-threitol, Pharmalyte pH 3–10 L (Pharmacia, Uppsala,

Sweden), supplemented with protease inhibitors

(com-plete, Boehringer Mannheim, Germany) The material

was then homogenized with an Ultraturrax (3· 10 s) and

further sonicated for 3· 10 s The homogenate was then

allowed to stand at room temperature for 60 min prior to

ultracentrifugation at 80 000 g at 15C for 1 h Aliquots of

the clear supernatant were frozen in liquid nitrogen and

stored at)80 C For determination of the protein

concen-tration, protein was precipitated with 10% trichloroacetic

acid in acetone and resuspended in 0.1MNaOH Protein

concentration was then determined with the Bio-Rad

protein assay (BIO-RAD, Int)

Two-dimensional PAGE

Protein (150 lg, for silverstained gels) and 600 lg protein

(for Coomassie-Blue staining) were loaded for each sample

onto the first dimension strips A linear immobilized pH

gradient (pH 5.0–6.0 IPG 18 cm; Pharmacia, Uppsala, Sweden) was used as the first dimension Hydration of gel strips and sample application was performed at 50 V for

15 h For protein separation a step voltage protocol was applied (1 h 150 V, 3 h 500 V, 1 h 1000 V, gradient

to 8000 V within 0.5 h) A total volt-hour product of

60 kVh was used for 150 lg protein and 110 kVh for

600 lg protein Afterwards the stripes were incubated in

50 mmolÆL)1Tris/HCl (pH 6.8), urea 6 molÆL)1, glycerol 30%, dithiothreitol 65 mmolÆL)1, 2% SDS for 20 min at room temperature followed by incubation in 50 mmolÆL)1 Tris/HCl (pH 8.8), urea 6 molÆL)1, glycerol 30%, iodo-acteamide 140 mmolÆL)1, 2% SDS for another 20 min For the second dimension, a vertical gradient slab gel of 8%)18% acrylamide was used and SDS/PAGE was performed at 8 mA per gel at 13C for 4 h followed by

30 mA for 12 h At the end of the second dimension, the gels were removed from the glass plates

Staining of two-dimensional PAGE The gels were fixed and stained with silver according to standard protocols [15] The gels were then scanned (Image Scanner Sharp JX-330, Amersham Biosciences) and ana-lysed with theIMAGE3.1 analysis software package (Amer-sham Bioscience) Each spot was matched from one gel

to another and the relative volume of matched spots was compared For preparative protein analysis higher amounts

of protein were loaded for two-dimensional PAGE and the protein spots were then stained with colloidal Coomassie-Blue

Protein sequence analysis Coomassie-Blue stained spots were excised from the gels and were subjected to ESI-MS analysis [16] Sequences obtained with ESI-MS analysis were then compared with the mouse and rat subset of the NCBInr.fasta protein database

cDNA cloning From the protein sequence of the obtained peptides the coding DNA sequence was obtained with database stand-ard programs A pair of sense primer 5¢-CGGGATCC TGCGAGCTACAAGTATTC-3¢ and antisense down-stream primer 5¢-GGAATTCTCCACATACTCAGCA TCG-3¢ was then used for standard RT-PCR cloning of a cDNA fragment of the sequenced protein A 192-bp cDNA fragment with the sequence: 5¢-tccacatactcagcatcgggggactg tatgtcatcaacttcacagaagctgtctgaagaatcatcgtgtttcgtccactgaaga acagccttctgggtctcctcactcagagattcgtccactgctccgagccgctcagcct gctcacactcctcaaggaggttggcttccttggaatacttgtagctcgca-3¢ was obtained This sequence was then further used for sequence comparisons and to generate a cRNA probe for RNase protection

RNA isolation Total RNA was extracted from freshly harvested cells and from frozen tissues according to the protocol of Chom-czynski and Sacchi [17]

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RNase protection assay of ERO1-L(a), adrenomedullin

(ADM) and b-actin mRNA

ERO1-L(a), ADM and b-actin mRNA levels were

meas-ured by RNase protection assay as described previously

[18] In brief, radiolabelled antisense cRNA probes were

synthesized by in vitro transcription of plasmid vectors

carried subcloned cDNA fragments for ERO1-L, ADM

and b-actin with SP6 polymerase (Promega) in the presence

of [a-32P]GTP (Amersham) Labeled cRNA probes were

hybridized with total RNA at 60C for 16 h, then digested

with RNase A/T1 at room temperature for 30 min and

proteinase K at 37C for 30 min After phenol/chloroform

extraction and ethanol precipitation, the protected RNA

hybrids were separated by electrophoresis on 8%

polyacryl-amide gels After drying the gels, the amount of

radio-activity was assessed by an Instant Imager (Packard) in

counts per minute (c.p.m.) and autoradiography was

performed at)80 C for 1 day Results were expressed as

in proportion to b-actin mRNA as internal standard

Real time PCR analysis of mouse and human ERO1-La

and ERO1-Lb mRNA and b-actin mRNA

Real time PCR was performed in a Light Cycler (Roche,

Germany) All PCRexperiments were performed using the

Light Cycler DNA Master SYBRGreen I kit provided by

Roche Molecular Biochemicals (Mannheim, Germany)

Each reaction (20 lL) contained 2 lL cDNA, 3.0 mM

MgCl2, 1 pmol of each primer and 2 lL of Fast Starter

Mix (containing buffer, dNTPs, SYBRGreen and hotstart

Taq polymerase) The following primers were used For

human ERO1-La (gi|6272556); sense primer: 5¢-CGGGAT

CCTGATGAAGTTCCTGATGG-3¢, antisense primer:

5¢-GGAATTCGTCTGTGGCTTAAAACAG-3¢ For

human ERO1-Lb (gi|9716556); sense primer: 5¢-CGGGAT

CCCTGGGCAAGATATGATGA-3¢, antisense primer:

5¢-GGAATTCATTGATGCTAGCATGAAG-3¢ For

mouse ERO1-La (gi|15718668); sense primer: 5¢-CGGGA

TCCTGCGAGCTACAAGTATTC-3¢, antisense primer:

5¢-GGAATTCGCCACATACTCAGCATCg-3¢ For

mouse ERO1-Lb (gi|19744822); sense primer: 5¢-CGG

GATCCCTTTTGTGAACTTGATGA-3¢, antisense

pri-mer: 5¢-GGAATTCAGCCACGTATAGAATGAt-3¢

For mouse and human b-actin (gi|6671508); sense primer:

5¢-CGGGATCCCCGCCCTAGGCACCAGGGTG-3¢,

antisense primer: 5¢-GGAATTCGGCTGGGGTGTTGA

AGGTCTCAAA-3¢

The amplification program consisted of 1 cycle at 95C

for 10 min, followed by 40 cycles with a denaturing phase at

95C for 15 s, an annealing phase of 5 s at 60 C and a

elongation phase at 72C for 15 s A melting curve analysis

was performed after amplification to verify the accuracy of

the amplicon For verification of the correct amplification,

PCRproducts were analyzed on an ethidium bromide

stained 2% agarose gel

In each real-time-PCRrun for ERO1-L and for b-actin a

calibration curve was included, that was generated from

serial dilutions (1 : 1, 1 : 10, 1 : 100, 1 : 1000) of a cDNA

generated from the pooled RNA of the normoxic (control)

cultures (time 0) of the respective experimental series

(standard cDNA) Analysis of the individual unknowns

therefore yielded values relative to this pool Data are presented as the relative ERO1-L mRNA/b-actin mRNA ratio The ERO1-L mRNA/b-actin mRNA ratio of the standard (pool) cDNA was set to 1.0 (i.e time 0)

Statistics Levels of significance between groups were calculated using ANOVAtest followed by Bonferoni’s reduction for multiple comparisons P < 0.05 was considered significant Results

Screening the rat vascular smooth muscle cell line A7r5 for hypoxia induced proteins by 2D-electrophoresis revealed a highly reproducible and marked (about 20-fold) upregulated abundance of a protein with an pI of around pH 5.7 and

an apparent molecular mass of 58 kDaA on SDS/PAGE (Fig 1) By ESI-MS tryptic peptides were identified that covered 45.9% of the aminoacid sequence of the mouse ERO1-like protein, which consists of a total of 464 amino acids (gi|7657067) Based on the sequenced peptides a

Fig 1 2D-electrophoresis of proteins isolated from the rat vascular smooth muscle cell line, A7r5 kept at either 21% O 2 (A) or 1% O 2 (B) for 12 h Note the upregulation of the indicated protein spot.

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cDNA fragment was cloned by RT-PCR standard

tech-niques The resulting 192 bp cDNA sequence shared a

100% homology with rat ERO1-1(gi|18250365), 88%

homology with mouse ERO1-La (gi|15718668), 85%

homology with human ERO1-La (gi|7021225), but no

significant homology with human ERO1-Lb (gi|9845248)

or mouse ERO1-Lb (gi|19744822)

It was concluded therefore that the cloned cDNA was rat

ERO1-L(a) cDNA and the hypoxia induced protein was rat

ERO1-L(a) [rER O1-L(a)] The cloned cDNA was then

used to generate cRNA probes for quantification of

rERO1-L(a) mRNA by RNAse protection

It turned out that the abundance of rERO1-L(a) mR NA

in A7r5 cells at high oxygen tensions (21% O2) was rather

low, but increased strongly (20-fold) with a characteristic

time pattern and reached a stable plateau level after

exposure of the cells to low oxygen tensions (1% O2)

(Fig 2, upper panel)

A next set of experiments was designed to test for the

in vivorelevance of the findings obtained with A7r5 cells

For this goal rats were exposed either to room atmosphere

(21% O2) or to a low inspiratory oxygen tension (8% O2)

and rERO1-1(a) mRNA was semiquantitated by RNAse

protection in the different organs As shown in Table 1

rERO1-L(a) mRNA was upregulated by hypoxia in all

organs examined, except the brain, in which only a marginal

increase was found To determine whether the upregulation

of rERO1-L(a) was not only related to a fall of the arterial oxygen tension but more generally to a fall of cellular oxygen tension, we also examined the effect of carbon monoxide (CO) inhalation [0.1%] 0.1% CO inhibits oxygen transport by hemoglobin by about 50% and thus diminishes oxygen delivery to the tissues without changing arterial oxygen tension Depending on the rate of tissue oxygen consumption CO will therefore lower tissue oxygen tension

It turned out that also CO clearly stimulated rERO1-L(a) mRNA levels in the different rat organs, with the exception

of the lung, in which tissue oxygen tensions are directly related to inspiratory oxygen tensions rather than to the oxygen carrying capacity of the blood (Table 1) Thus, the failure of CO to stimulate rERO1-L(a) expression in the lung, can be taken as an argument that CO did not itself increase rERO1-L(a) expression rERO1-L(a) in vivo was also stimulated by the divalent cation cobalt, that was subcutaneously administered [Table 1]

The temporal pattern of rERO1-L(a) mR NA in rat A7r5 cells was very similar to that of classic oxygen regulated genes, such as adrenomedullin (ADM) (Fig 2, lower panel), the expression of which is triggered by the hypoxia inducible transcription factor HIF-1 [19] In addition, rERO1-L(a) mRNA was, like ADM mRNA, upregulated by the divalent cation cobalt (100 lmolÆL)1) and by the iron chelator deferoxamine (100 lmolÆL)1) (Fig 3)

Hypoxia and deferoxamine also increased ERO1-La mRNA in the mouse hepatoma cell line Hepa1 (Fig 4), suggesting a species independent stimulatory effect of hypoxia on ERO1-La gene expression In contrast, in the mutant cell line Hepa1C4, which is unable to generate active HIF [13], hypoxia and deferoxamine failed to increase ERO1-La mRNA (Fig 4) within the first five hours Only after 12 h of hypoxia or incubation with deferoxamine ERO1-La mRNA increased moderately

Using Hepa1 cells we also examined the effect of hypoxia and desferoxamine on the abundance of ERO1-Lb mRNA

As shown in Fig 5 there was no change of ERO1-Lb mRNA after 4.5 h, when ERO1-La mRNA levels had already clearly increased After 12 h of hypoxia ERO1-Lb mRNA was moderately elevated In view of the different temporal response of ERO1-La and ERO1-Lb mRNA to hypoxia in mouse Hepa1 cells, we analyzed the early hypoxic response also in the mouse renal renin secreting As4.1 cell line [14] and in the human hepatoma Hep G2 cell

Fig 2 Time course of rERO1-L mRNA (upper panel) and of

adreno-medullin mRNA (lower panel) in A7r5 cells after exposure of the cells to

1% O 2 Data are means ± SEM of five experiments *Indicates

P < 0.05 hypoxia (1% O ) vs normoxia (21% O ).

Table 1 Effect of hypoxia (8% O 2 ), carbon monoxide (0.1%) inhala-tion and of administrainhala-tion of 60 mgÆkg)1 cobaltous chloride on ERO1-La mRNA in various rat tissues Results are presented as ratio ERO1-L(a) mR NA/b-actin mRNA · 10 2

Data are means ± SEM

of 4–6 animals *Indicates P < 0.05 vs 21% O 2

Organ 21% O 2 8% O 2 0.1% CO

Cobaltous chloride (60 mgÆkg)1) Aorta 7 ± 1 14 ± 3* 16 ± 4* 11 ± 4 Brain 5.6 ± 1.6 6.5 ± 0.5 11.2 ± 1.4* 9.6 ± 2.1* Heart 8 ± 1 15 ± 2* 23 ± 4* 19 ± 5* Kidney 12 ± 4 57 ± 21* 31 ± 8* 25 ± 4* Liver 20 ± 8 110 ± 28* 350 ± 30* 230 ± 30* Lung 1.8 ± 0.3 2.9 ± 0.5* 1.9 ± 0.2 3.8 ± 1.1*

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line It turned out that 4.5 h of hypoxia induced ERO1-La

but not ERO1-Lb mRNA (Fig 6) Similar results were

obtained for the effect of deferoxamine

As a differential regulation of ERO1-La and ERO1-Lb

mRNA expression has been reported previously, in the way

that the unfolded protein response (UPR) pathway

prefer-entially induces ERO1-Lb mRNA expression [7], we aimed

to examine this concept in our model of mouse As4.1 cells

We found, that tunicamycin (5 lgÆmL)1), which induces the

UPR, increased ERO1-La and ERO1-Lb mRNA about

fourfold after 4.5 h of incubation Whilst ERO1-Lb mRNA

further increased to a plateau 12-fold above control,

declined ERO1-La mRNA after prolonged incubation to

reach a plateau twofold over control (Fig 7)

Discussion

Correct protein folding in the endoplasmic reticulum

essentially requires the activity of the protein disulfide

isomerase PDI, which in turn is dependent on the delivery

of oxidizing equivalents by endoplasmic oxidoreductase

ERO1, which occurs in an La- and in a Lb-isoform in

mammals ERO1-L isoforms in conjunction with PDI

therefore fulfil chaperone function It is well known that a

variety of endoplasmic proteins with chaperone function are

induced by energy depletion caused by severe cellular

hypoxia (anoxia) or by glucose deprivation [20] It is

thought that the expression of these proteins in response to anoxia is triggered by the unfolded protein response (UPR) which regulates the activity of chaperone genes [21] and leads to attenuation of protein synthesis via the activation of the endoplasmic reticulum kinase PERK [22] Unfolding or misfolding of proteins in the endoplasmic reticulum during anoxia probably results from ATP depletion and also from changes of redox potentials In consequence, yeast ERO1 [3] and ERO1-L b in human tissues [7] are also stimulated by UPR Interestingly, ERO1-L a appears to be less affected by UPR[7] suggesting that ERO1-L a is differently regulated

in its expression

Our data now indicate that the expression of the rat, mouse and human isoform of ERO1-L(a) is strongly upregulated following a decrease in the cellular oxygen tension Apparently, this phenomenon appears to be of major relevance also under in vivo conditions under which rERO1-L(a) expression is also markedly increased during hypoxia Our data also show that not only arterial hypoxia but also a reduction of the oxygen carrying capacity of the blood (by CO inhalation) stimulates rERO1-L(a) gene expression in various tissues

Our data provide several lines of evidence to suggest that the expression ERO1-La is probably triggered by the hypoxia-inducible transcription factor (HIF-1)

Fig 3 rERO1-L(a) mRNA (upper panel) and adrenomedullin mRNA

(lower panel) in A7r5 cells after exposure to 0.5% O 2 or to cobaltous

chloride (100 lmolÆL)1) or deferoxamine (100 lmolÆL)1) for 12 h at

21% O 2 Data are means ± SEM of five experiments each *Indicates

P < 0.05 vs control (21% O 2 ).

Fig 4 Mouse ERO1-La mRNA in Hepa1 (upper panel) and in Hepa1C4 cells (lower panel) after exposure to hypoxia (0.5% O 2 ) (100 lmolÆL)1) or to deferoxamine (100 lmolÆL)1) at 21% O 2 mRNA was semiquantitated by real-time PCR Data are means ± SEM of five experiments each *Indicates P < 0.05 vs control (21% O 2 ).

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HIF-1 is a heterodimer consisting of an a- and a

b-subunit [23] HIF-1a stability is regulated by the cellular

oxygen tension, in the way that an oxygen/iron dependent

prolyl-hydroxylation leads to increased ubiquitinylation

and finally proteasomal degradation of HIF-1a [24,25] In

consequence, a decrease of prolyl-hydroxylase activity by

low oxygen tensions, by iron chelation or by cobalt increase

HIF-1a protein levels and therefore the activity of the

HIF-1 transcription factor [26]

The temporal pattern of the induction of rERO1-L(a)

expression by hypoxia in vitro is very similar to HIF-1

regulated genes, such as adrenomedullin [19] Moreover, the

effect of hypoxia on ERO1-La gene expression can be

mimicked in a very characteristic fashion by cobalt and by

the iron chelator deferoxamine, which do not change

cellular oxygen tension but increase HIF-1a and therefore

stimulate HIF-1 activity [27,28] Finally, the early

stimula-tion of ERO-1a gene expression was absent in a cell line

with a functional mutation in the HIF-1b gene, which

causes an inability to form active HIF [13] The moderate

increase of ERO-1a gene expression in HIF deficient cells

after prolonged hypoxia is probably explained by unfolded

protein response pathway, which is evoked by prolonged

hypoxia and which itself moderately triggers ERO1-La

gene expression as seen in this study [Fig 7]

In contrast to ERO-1a gene expression, ERO1-Lb mRNA was not upregulated by acute hypoxia in the mouse and human cell lines, suggesting that hypoxia per se is not a major trigger for ER O1-Lb gene expression The moderate

of increase of ERO1-Lb mRNA by prolonged hypoxia may

be again explained by the induction of the unfolded protein response, what would well fit with the concept that the UPR mainly triggers the ERO1-Lb gene [7]

The conclusion that EROl-La but not ERO1-Lb is triggered by HIF-1 is indirectely supported by the occurence

of the most common active HIF-binding consensus sequence ACGTG in the ERO1-L gene promotors Thus, rat, mouse and human EROl-La contain two, two and one ACGTC motifs in CpG islands in the 5¢-promoter region, respectively, whilst ERO1-Lb does not contain this motif

in GpC islands

HIF-1 regulated genes identified so far encode proteins that mainly serve to match the cellular energy deficit resulting from insufficient oxygen supply [29] Thus, glucose transporters and key enzymes of the glycolytic pathway are regulated by HIF-1 and are upregulated during hypoxia Also secreted proteins such as erythropoietin which stimu-lates red cell formation (and thus increases the oxygen carrying capacity of the blood) or vascular endothelial growth factor (VEGF), which induces capillary formation,

Fig 6 ERO1-La and ERO1-Lb mRNA in mouse As4.1 cells (upper panel) and in human HepG2 cells (lower panel) after exposure to hypoxia (0.5% O 2 ) (100 lmolÆL)1) or to deferoxamine (100 lmolÆL)1) at 21%

O 2 after 4.5 h of incubation mRNA was semiquantitated by real-time PCR Data are means ± SEM of five experiments each *Indicates

P < 0.05 vs control (21% O 2 ).

Fig 5 Mouse ERO1-La (upper panel) and ERO1-Lb mRNA (lower

panel) in Hepa1 (upper panel) cells (lower panel) after exposure to

hypoxia (0.5% O 2 ) or to deferoxamine (100 lmolÆL)1) at 21% O 2

mRNA was semiquantitated by real-time PCR Data are means ±

SEM of five experiments each *Indicates P < 0.05 vs control

(21% O 2 ).

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or adrenomedullin (ADM), which causes vasodilation, are

stimulated by HIF-1 in response to hypoxia (reviewed in

[29])

With the regulation of proteins that are involved in

correct folding of proteins in the endoplasmic reticulum,

HIF-1 would aquire a new responsibility for cellular

function (Fig 8) A regulation of ERO1-La production

by HIF-1 means that chaperone formation during hypoxia

is uncoupled from energy depletion (which initiates the

UPR), and thus allows a counterregulation in situations in

which the cellular redox state is already altered whilst the

energy state is still normal A number of endo- or paracrine

signals involved in the hypoxia defense such as for example

erythropoietin [30], VEGF [31] or ADM [32] in fact contain

disulfide bonds that are indispensable for their biological

function Problems with disulfide bond formation during a

fall of the oxygen tension may arise from the change of the

redox potential of the cell, which impairs the flow rate of

oxidizing equivalents from ERO1-L to PDI Under

redu-cing conditions PDI would actually catalyze the reduction

of protein disulfides [1] The relevance of PDI in this context

was underlined previously by the finding that

overexpres-sion of PDI attenuated the loss of cell viability induced by

hypoxia in a neuroblastoma cell line [33] As ERO1-La

exists as a collection of oxidized and reduced forms [9]

increasing the total number of ERO1-La molecules during

hypoxia would therefore compensate for the diminuation of

the redox gradient and maintain a constant flow of oxidizing equivalents to PDI over a broad range of cellular oxygen tension

The oxygen regulation of ERO1-La expression appears

to be part of a more general network in which the expression

of chaperones is regulated by the oxygen tension through HIF-1 Thus, it was shown previously that hypoxia increases the expression of PDI itself in brain cells in vitro and in vivo [33], although it was not further examined in that study as to whether the upregulation of PDI was mediated

by UPRor by the HIF-1 pathway PDI also serves as the b-subunit of the collagen prolyl-4-hydroxylase, which is a heterotetramer consisting of 2a and 2b subunits [34] It was reported previously for cultured fibroblasts that hypoxia induces the expression of a-subunit of the collagen prolyl-4-hydroxylase (I) through the HIF-1 pathway [35] All together, our findings suggest that a fall of the cellular oxygen tension compensatorily increases the expression of a protein that is required to transfer oxidizing equivalents to PDI, and is therefore required for correct protein folding in the endoplasmic reticulum

Acknowledgements

The authors thank K-H Go¨tz for doing the artwork and Vladimir Todorov for helpful discussions.

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Fig 8 Summary of the regulation of ERO1-L expression by the oxygen tension and by the unfolded protein response Abbreviations: pO 2 , cel-lular oxygen tension; ATP, concentration of adenosine triphosphate; ERendoplasmic reticulum; UPR , unfolded protein response; PDI, protein disulfide isomerase.

Fig 7 ERO1-La and ERO1-Lb mRNA in mouse As4.1 cells after

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